Challenges to achieving low palatal fistula rates following primary cleft palate repair: experience of an institution in Uganda
Katusabe et al. BMC Res Notes
Challenges to achieving low palatal fistula rates following primary cleft palate repair: experience of an institution in Uganda
Josephine Linda Katusabe 0
Andrew Hodges 0
George William Galiwango 0
Edgar M. Mulogo
0 Comprehensive Rehabilitation Services Uganda (CoRSU) Hospital , P.O.Box 46, Kisubi , Uganda
Objective: To determine frequency of palatal fistula following primary cleft palate repair and the associated factors as a measure of cleft palate repair outcome and its challenges at a cleft centre in Uganda. Results: Between May and December 2016, 54 children with cleft palate were followed up at Comprehensive Rehabilitation services of Uganda (CoRSU) hospital, from time of primary cleft palate repair until at least 3 months postoperative to determine whether they developed palatal fistula or not. Frequency of palatal fistula was 35%. Factors associated with increased fistula formation were cleft width wider than 12 mm (p = 0.006), palatal index greater than 0.4 (p = 0.046), presence of malnutrition at initial outpatient assessment (p = 0.0057) and at time of surgery (p = 0.008), two-stage palate repair (p = 0.005) and postoperative infection (p = 0.003). Severe clefting (palatal index greater than 0.4) was seen in 74% of patients and malnutrition (Low weight for age) seen in 48% of patients. Palatal fistula rates at our institution were high compared to reports in literature. The high proportions of severe clefting and malnutrition observed in our population that was also poor and unable to afford feeding supplements increased likelihood of fistula formation and posed challenges to achieving low fistula rates in our setting.
Cleft palate; Primary palate repair; Palatal fistula
Palatal fistula is a failure of healing or breakdown in the
primary surgical repair of a cleft palate [
fistula results in persistent communication between oral
and nasal cavities leading to unpleasant symptoms such
as nasal spillage of feeds, hypernasal speech, articulation
problems which undermine the success of palate repair
]. A low incidence of palatal fistula is one of the
indicators of successful cleft palate repair [
Incidence of palatal fistula in literature ranges from 0 to
] with overall incidence of 8.6% reported by
a meta-analysis of studies in Europe, America, Asia and
]. Risk factors of palatal fistula reported include
type of cleft, cleft palate width, surgeon’s experience,
timing and technique of repair. There is a paucity of studies
in Africa and Uganda assessing frequency of palatal
fistula and associated factors following cleft palate repair. In
Uganda, reports show that most children with cleft palate
are already malnourished when they first present to
hospital and may continue failing to thrive if no timely
intervention is done [
]. Effect of this malnutrition on
surgical outcome of palate repair has not been studied.
Our study aimed to determine frequency of palatal
fistula following primary palate repair and the associated
factors at CoRSU hospital in order to assess our cleft
palate repair outcome and also establish the challenges to
achieving low fistula rates.
A prospective case series was conducted from May to
December 2016 at CoRSU hospital, a specialized
hospital in Uganda offering free cleft palate surgery. Children
with unrepaired cleft palate, whose caregivers gave
written consent to participate in the study were enrolled and
followed up from time of primary cleft palate repair until
at least 3 months postoperative to determine whether
they developed palatal fistula or not. Primary
palate repair (surgery on cleft palates that have not been
repaired before) was performed either as single-stage
repair of both hard and soft palate or as a two-stage
procedure involving hard palate repair with vomer flap in
first stage and soft palate repair 3 months later in second
stage. Surgical techniques included intravelar veloplasty
for soft palate, Von Langenbeck flaps, Bardarch flaps and
Hybrid flaps for hard palate (see Additional file 1 showing
description of surgical techniques).
Desired perioperative information including age,
weight and length, type of cleft, type of surgery, surgical
technique and surgeon’s experience (based on volume
of palate surgeries performed annually) was recorded in
pretested data forms. Preoperative dental casts of each
palate were made, from which cleft width and palatal
shelf widths were measured using Castroviejo calipers
(see Additional file 2 showing dimensions measured).
Weight for length and weight for age z-scores were
calculated and compared with W.H.O reference values to
determine nutrition status. At postoperative review, a
consultant plastic surgeon inspected the palate using a
clinical torch and tongue depressor to determine
presence or absence of fistulas. Only fistulas posterior to
incisive foramen were considered. Statistical analysis of
data was done using STATA version 12.0. Chi square test,
student t test, and multivariate logistic regression were
performed to determine factors associated with palatal
fistula. Probability values (p-values) less than 0.05 were
considered statistically significant.
A total of 78 children were enrolled but only 54 patients
followed up at CoRSU hospital were analysed. Median
age was 6 months (range 3–192 months) with 61% of
participants below 6 months. Female to male ratio was 1.1:1.
Unilateral cleft lip and palate was seen in 52%, bilateral
cleft lip and palate in 37% and isolated cleft palate in 11%
of patients. Most of the caregivers (61%) resided in rural
settings with median monthly income of 28 US dollars.
Forty-eight percent (48%) of patients had low weight for
length at initial outpatient assessment and 62% of these
received nutrition rehabilitation before surgery. Median
weight at surgery was 5.7 kg (range 3.5–50 kg).
Twentyfour percent (24%) were underweight (low weight for
age) and 15% were severely underweight at time of
surgery despite attaining minimum required weight for
surgery (3.5 kg). Mean cleft width was 12 mm with 48% of
patients having clefts wider than 12 mm. Mean ratio of
cleft width to sum of palatal shelf width (palatal index)
was 0.4 with 74% of patients having palatal index greater
Single-stage repair was done in 67% of patients while
33% had two-stage repair. Vomer flap dehiscence
occurred in 67% of two-stage repairs and required hard
palate re-repair at second stage. Large-volume operator
(surgeon performing over 50 palate surgeries annually)
performed 67%, low-volume operator (surgeon
performing less than 50 palate surgeries annually) performed 24%
and trainee surgeon performed 9% of the surgeries. Von
Langenbeck flaps were used in 52%, Bardarch in 22% and
hybrid in 26% of patients.
Postoperative complications included early
postoperative infection estimated as persistent fever with
leukocytosis requiring antibiotics (56%), difficulty in breathing
requiring oxygen (38%) and flap dehiscence (6%). 37% of
participants reported late postoperative infection
estimated as falling sick within 4 weeks after discharge and
60% of these required admission to a hospital near home.
Average follow up period was 6 months.
Overall frequency of palatal fistula was 35% (95% CI
22.4, 47.9) with 19 out of 54 patients developing palatal
fistula. Twenty-eight percent (15 out of 54) had fistulas
requiring surgical repair. Hard palate (Pittsburgh IV) was
involved in 58%, Soft palate (Pittsburgh II) involved in
25% and junction of hard and soft palate (Pittsburgh III)
involved in 17% of fistula. (See Additional file 2 showing a
case with hard palate fistula).
Factors associated with increased fistula formation
were: Low weight for length at initial outpatient
assessment (p = 0.0057), low weight for age at time of surgery
(p = 0.008), cleft width wider than 12 mm (p = 0.006),
palatal index greater than 0.4 (p = 0.046), two-stage
palate repair (p = 0.005), early postoperative infection
(p = 0.003) and late postoperative infection (p = 0.0004)
as shown in Tables 1, 2 and 3. Cleft width was an
independent predictor of palatal fistula at multivariate
analysis (p = 0.03, Adjusted odds ratio = 4.4). No association
was found between age, surgical technique, surgeon’s
experience and fistula formation.
Frequency of palatal fistula at our institution is high
compared to reports in literature [
] and loss to
follow up, difference in population characteristics could be
responsible for this discrepancy. Loss to follow up could
have resulted in artificially higher fistula rates if some of
the patients lost actually had no fistulas. Patients
without palatal fistulas don’t experience the related
unpleasant symptoms that would otherwise prompt seeking of
further treatment and follow up. Loss to follow up could
also have been due to low socioeconomic status where
most of the caregivers were poor (earning 28 US dollars
a month), lived in remote areas and probably could not
afford transport to hospital for review. This possibly
hindered return of both patients with and without fistulas
resulting in under or overestimation of our fistula rates.
The proportion of malnutrition in our population was
high, both at initial outpatient assessment and at time of
surgery and this probably contributed to the high fistula
rates. Results actually showed that low weight for length
at initial outpatient assessment and low weight for age
at time of surgery were associated with increased fistula
formation (Table 1). Malnutrition impedes processes that
allow progression of wound healing and has been related
to decreased wound tensile strength and increased
infection rates [
]. Patients with malnutrition are therefore
prone to wound breakdown following surgery which
explains their increased likelihood for fistula formation.
Prevalence of malnutrition among children with
cleft palate in Africa has been reported to be high by
other studies [
11, 12, 18, 19
]. The trend is that most
children with cleft palate present with chronic
malnutrition and stunting and many are hypothesized to die
before surgery due to malnutrition . Intervention
through nutritional rehabilitation and surgery has been
recommended to improve their survival [
malnutrition with stunting requires prolonged periods
of nutritional rehabilitation before optimal nutrition
status can be attained but majority of our patients are
poor and cannot afford feeding supplements. This
creates a dilemma for the cleft surgeon trying to achieve
good surgical outcomes in a setting where optimizing
nutrition status before surgery is difficult. During the
study period, our centre performed palate repair as
early as 3 months, at minimum weight of 3.5 kg, with
the aim of improving feeding. With this protocol, some
of the children were still underweight at surgery yet it
hasn’t been demonstrated that surgery in underweight
children with cleft palate actually improves their
nutrition status hence there is need for more research.
Palatal index greater than 0.4 is classified as severe
clefting with significant tissue deficiency [
]. A high
proportion of patients had clefts wider than 12 mm
(48%) and palatal index greater than 0.4 (74%)
indicating that most of our study population had wide and
severe clefting, that was actually found to be
associated with increased fistula formation(Tables 1 and 2).
Other studies have also shown that cleft width wider
than 13–15 mm and palatal index are associated with
increased fistula formation [
]. According to
Parwaz et al., the risk of fistula formation increases as
palatal index increases to 0.48 . Wide clefts are prone to
tension on closure and are related with more technical
difficulties to close than narrow clefts which explains
why patients with wide clefts are more likely to develop
Two-stage palate repair was associated with increased
fistula formation in our study (Table 3). This is contrary
to other studies that reported significant reduction in
cleft width, operating time and fistula formation
following early hard palate repair using vomer flap [
The high rate of vomer flap dehiscence possibly
contributed to the increased fistula formation in the two-stage
group. Vomer flap dehiscence required re-repair of hard
palate at second stage using previously scarred and less
pliable vomer tissue that was of poor quality and prone
to breakdown. This issue was also raised by Deshpande
et al. who found that failed vomer flaps increased risk
of fistula formation in subsequent palate repairs .
More research is needed to investigate causes of vomer
flap failure in our setting which if addressed could help
reduce fistula formation. Our centre performs vomer
flaps for clefts deemed too wide to close in a single stage,
to help reduce cleft width while also providing an intact
hard palate to improve suckling. Effectiveness of this
two-stage protocol in reducing cleft width and improving
nutrition status in our patients with severe clefting and
malnutrition needs to be studied to help justify its use in
treating wide clefts.
Early and late postoperative infection was a common
complication in our study and was associated with palatal
fistula formation (Table 3). Postoperative infection is one
of the likely reasons for fistula formation but is unlikely
in babies unless compromised immunologically or
]. The high proportion of malnutrition seen
in our population that mainly comprised babies below
6 months could have predisposed to increased
postoperative infection which probably translated into higher
Frequency of palatal fistula at our institution was high.
The high proportions of severe clefting and
malnutrition in our population that was also poor and unable to
afford feeding supplements increased likelihood of
fistula formation and posed challenges to achieving low
fistula rates in our setting. We recommend more efforts
on optimizing nutrition status before surgery through
nutritional education and feeds supplementation, even
in the face of a challenging low socioeconomic status.
More research is needed to determine effect of surgery
on nutrition status of children with cleft palate which will
help to guide better the timing of surgery with regard to
nutrition status. Further research is also needed to
investigate effectiveness of vomer flap in reducing cleft width
and improving nutrition status to help justify its benefit
in treating wide clefts in our population.
• Lack of funds hindered return of some participants
for review resulting in loss to follow up which could
have resulted in over or underestimation of fistula
• Study was done at a single surgical centre in Uganda
with a small sample size which may not adequately
represent palate repair outcome in other Ugandan
Additional file 1. Description of surgical techniques used for primary
palate repair in our study. Brief descriptions of the surgical techniques
used for palate repair is provided. These include, Von Langenbeck flap,
Bardach flap, Hybrid flap and vomer flap techniques for hard palate repair
and intravelar veloplasty for soft palate repair.
Additional file 2. A Figure showing a hard palate fistula and
dimensions of the cleft palate that were measured. Figure 1a) shows a case
with a hard palate fistula (Pittsburgh IV). In Figure 1b) distance B-C is
cleft width measured at junction of hard and soft palate, A-B and C-D are
the right and left palatal shelf widths measured at level of the maxillary
KJL: was the principal investigator in the study who designed and conducted
the study, analysed and interpreted the data and wrote the manuscript. AH,
GGW and EM: were the research supervisors providing overall guidance in
the research and also critically reviewed the manuscript. All authors read and
approved the final manuscript.
The author is a Plastic and reconstructive surgeon trained at Mbarara
University of Science and Technology (CoRSU hospital campus) and currently
working at CoRSU hospital in Uganda. The research study was done as a
requirement for the achievement of Masters of Medicine in Plastic and
Reconstructive surgery at the University.
1 Comprehensive Rehabilitation Services Uganda (CoRSU) Hospital, P.O.Box 46,
Kisubi, Uganda. 2 Mbarara University of Science and Technology, P.O.Box 1410,
Authors would like to thank all the staff at CoRSU hospital and Mbarara
University of Science and Technology (MUST) for their contribution towards
completion of this research.
The authors declare that they have no competing interests.
Availability of data and material
The datasets generated and analysed during this study are not publicly
available as the author is still using the data set to carry out more research.
Consent for publication
Figure 1a) and 1b) shows images of two study patients that were taken by
author KJL. Written consent was obtained from parents of these children for
the publication of these photos.
Ethics approval and consent to participate
Study was approved by the Mbarara University of Science and Technology
Research Ethics committee and Uganda National Council of Science and
Technology (UNCST). Written consent to participate was sought from the parents
and caretakers of children enrolled in the study.
Study was funded by Mbarara University of Science and Technology as part of
its policy to help fund research projects for students. It funded all aspects of
the study from proposal development to manuscript development.
Springer Nature remains neutral with regard to jurisdictional claims in
published maps and institutional affiliations.
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